Tracheoscopic Findings and Their Impact on Respiratory Symptoms in Children with Esophageal Atresia
05. Dezember 2018
07. Februar 2019
21. März 2019 (online)
Introduction Esophageal atresia (EA) is often accompanied by tracheobronchial malformations leading to stridor, recurrent bronchitis, and occasionally to life-threatening obstructive apnea after surgical repair. The aim of this study was to identify the presence of tracheomalacia in patients with EA and tracheoesophageal fistula (TEF) pre- and postoperatively and to find endoscopic correlates leading to clinical airway symptoms.
Methods In a cohort of 362 patients with EA-TEF who underwent 595 tracheoscopies at the Children's Hospital of Cologne between January 1983 and December 2002, impaired tracheal lumen, localization of TEF, tracheal pulsations, and corresponding clinical symptoms were retrospectively analyzed.
Results The incidence of tracheomalacia was higher in patients with EA and TEF (Gross B–D) compared with patients with EA alone (Gross A) and average tracheal collapse does not significantly change before and after surgical repair of the esophagus in all types. Patients with cyanosis while eating and obstructive apnea presented with an average tracheal collapse of 89%. The presence of respiratory symptoms such as cough, stridor, or bronchitis was not associated with a higher grade of tracheal collapse compared with patients without any airway symptoms (average tracheal collapse of 37% in symptomatic patients vs. 33% in nonsymptomatic patients).
Conclusion Tracheomalacia tends to be present independently of surgical procedure. Tracheomalacia should be measured by tracheoscopy (in % of tracheal collapse). Patients with a tracheal collapse of >80%, a ventral pulsation, and obstructive apnea or cyanosis in combination, are at risk for life-threatening situations and further surgical treatment should be considered.
Keywordstracheal collapse - tracheomalacia - laryngotracheobronchoscopy - tracheoesophageal fistula
* Janina Fischer and Johannes Balleisen contributed equally to the study.
- 1 Hseu A, Recko T, Jennings R, Nuss R. Upper airway anomalies in congenital tracheoesophageal fistula and esophageal atresia patients. Ann Otol Rhinol Laryngol 2015; 124 (10) 808-813
- 2 Dellenmark-Blom M, Chaplin JE, Gatzinsky V. , et al. Health-related quality of life experiences among children and adolescents born with esophageal atresia: development of a condition-specific questionnaire for pediatric patients. J Pediatr Surg 2016; 51 (04) 563-569
- 3 Malmström K, Lohi J, Lindahl H. , et al. Longitudinal follow-up of bronchial inflammation, respiratory symptoms, and pulmonary function in adolescents after repair of esophageal atresia with tracheoesophageal fistula. J Pediatr 2008; 153 (03) 396-401
- 4 Sistonen S, Malmberg P, Malmström K. , et al. Repaired oesophageal atresia: respiratory morbidity and pulmonary function in adults. Eur Respir J 2010; 36 (05) 1106-1112
- 5 Gatzinsky V, Wennergren G, Jönsson L. , et al. Impaired peripheral airway function in adults following repair of esophageal atresia. J Pediatr Surg 2014; 49 (09) 1347-1352
- 6 Chetcuti P, Phelan PD. Respiratory morbidity after repair of oesophageal atresia and tracheo-oesophageal fistula. Arch Dis Child 1993; 68 (02) 167-170
- 7 Little DC, Rescorla FJ, Grosfeld JL, West KW, Scherer LR, Engum SA. Long-term analysis of children with esophageal atresia and tracheoesophageal fistula. J Pediatr Surg 2003; 38 (06) 852-856
- 8 Gatzinsky V, Jönsson L, Ekerljung L, Friberg LG, Wennergren G. Long-term respiratory symptoms following oesophageal atresia. Acta Paediatr 2011; 100 (09) 1222-1225
- 9 Sharma N, Srinivas M. Laryngotracheobronchoscopy prior to esophageal atresia and tracheoesophageal fistula repair--its use and importance. J Pediatr Surg 2014; 49 (02) 367-369
- 10 Taghavi K, Stringer MD. Preoperative laryngotracheobronchoscopy in infants with esophageal atresia: why is it not routine?. Pediatr Surg Int 2018; 34 (01) 3-7
- 11 Benjamin B. Endoscopy in esophageal atresia and tracheoesophageal fistula. Ann Otol Rhinol Laryngol 1981; 90 (4 Pt 1): 376-382
- 12 Holzki J. Bronchoscopic findings and treatment in congenital tracheo-oesophageal fistula. Paediatr Anaesth 1992; 2: 297-303
- 13 Jacobs IN, Wetmore RF, Tom LW, Handler SD, Potsic WP. Tracheobronchomalacia in children. Arch Otolaryngol Head Neck Surg 1994; 120 (02) 154-158
- 14 Vazquez-Jimenez JF, Sachweh JS, Liakopoulos OJ. , et al. Aortopexy in severe tracheal instability: short-term and long-term outcome in 29 infants and children. Ann Thorac Surg 2001; 72 (06) 1898-1901
- 15 Gruszka A, Sachweh JS, Schnoering H. , et al. Aortopexy offers surgical options for a variety of pathological tracheal conditions in paediatric patients. Interact Cardiovasc Thorac Surg 2017; 25 (04) 589-594
- 16 Boogaard R, Huijsmans SH, Pijnenburg MW, Tiddens HA, de Jongste JC, Merkus PJ. Tracheomalacia and bronchomalacia in children: incidence and patient characteristics. Chest 2005; 128 (05) 3391-3397
- 17 Zani A, Eaton S, Hoellwarth ME. , et al. International survey on the management of esophageal atresia. Eur J Pediatr Surg 2014; 24 (01) 3-8
- 18 Atzori P, Iacobelli BD, Bottero S. , et al. Preoperative tracheobronchoscopy in newborns with esophageal atresia: does it matter?. J Pediatr Surg 2006; 41 (06) 1054-1057
- 19 Greemberg L, Fisher A, Katz A. Novel use of neonatal cuffed tracheal tube to occlude tracheo-oesophageal fistula. Paediatr Anaesth 1999; 9 (04) 339-341
- 20 Shoshany G, Vatzian A, Ilivitzki A, Smolkin T, Hakim F, Makhoul IR. Near-missed upper tracheoesophageal fistula in esophageal atresia. Eur J Pediatr 2009; 168 (10) 1281-1284
- 21 Usui N, Kamata S, Ishikawa S. , et al. Anomalies of the tracheobronchial tree in patients with esophageal atresia. J Pediatr Surg 1996; 31 (02) 258-262
- 22 Carden KA, Boiselle PM, Waltz DA, Ernst A. Tracheomalacia and tracheobronchomalacia in children and adults: an in-depth review. Chest 2005; 127 (03) 984-1005
- 23 Ngerncham M, Lee EY, Zurakowski D, Tracy DA, Jennings R. Tracheobronchomalacia in pediatric patients with esophageal atresia: comparison of diagnostic laryngoscopy/bronchoscopy and dynamic airway multidetector computed tomography. J Pediatr Surg 2015; 50 (03) 402-407
- 24 Spitz L, Kiely E, Brereton RJ. Esophageal atresia: five year experience with 148 cases. J Pediatr Surg 1987; 22 (02) 103-108
- 25 Slany E, Holzki J, Holschneider AM, Gharib M, Hügel W, Mennicken U. Tracheal instability in tracheo-esophageal abnormalities [in German]. Z Kinderchir 1990; 45 (02) 78-85
- 26 Kamran A, Hamilton TE, Zendejas B, Nath B, Jennings RW, Smithers CJ. Minimally invasive surgical approach for posterior tracheopexy to treat severe tracheomalacia: lessons learned from initial case series. J Laparoendosc Adv Surg Tech A 2018; DOI: 10.1089/lap.2018.0198.
- 27 Tytgat SHAJ, van Herwaarden-Lindeboom MYA, van Tuyll van Serooskerken ES, van der Zee DC. Thoracoscopic posterior tracheopexy during primary esophageal atresia repair: a new approach to prevent tracheomalacia complications. J Pediatr Surg 2018; 53 (07) 1420-1423